Symptomatic Brain Hemorrhages from Cavernous Angioma After Botulinum Toxin Injections, a Role of TLR/MEKK3 Mechanism? Case Report and Review of the Literature

Janne Koskimäki; Dongdong Zhang; Julián Carrión-Penagos; Romuald Girard; Kristina Piedad; Sean P Polster; Seán Lyne; Agnieszka Stadnik; Issam A Awad

doi:10.1016/j.wneu.2019.12.172

Symptomatic Brain Hemorrhages from Cavernous Angioma After Botulinum Toxin Injections, a Role of TLR/MEKK3 Mechanism? Case Report and Review of the Literature

World Neurosurg. 2020 Apr:136:7-11. doi: 10.1016/j.wneu.2019.12.172. Epub 2020 Jan 7.

Authors

Janne Koskimäki¹, Dongdong Zhang¹, Julián Carrión-Penagos¹, Romuald Girard¹, Kristina Piedad¹, Sean P Polster¹, Seán Lyne¹, Agnieszka Stadnik¹, Issam A Awad²

Affiliations

¹ Neurovascular Surgery Program, Section of Neurosurgery, The University of Chicago Medicine and Biological Sciences, Chicago, Illinois, USA.
² Neurovascular Surgery Program, Section of Neurosurgery, The University of Chicago Medicine and Biological Sciences, Chicago, Illinois, USA. Electronic address: iawad@uchicago.edu.

PMID: 31917316
DOI: 10.1016/j.wneu.2019.12.172

Abstract

Background: Cavernous angiomas (CAs) are vascular malformations that may result in stroke.

Case description: Herein, we evaluate a CA patient with chronic migraine who experienced 2 documented symptomatic hemorrhages after receiving respective high doses of botulinum toxin (Btx).

Conclusions: Recently, bacterial lipopolysaccharide has been reported to contribute to CA development through Toll-like receptor signaling, causing hemorrhagic angiogenic proliferation. Lipopolysaccharide and Btx share a common intracellular signaling pathway driving CA development and hemorrhage. Significance of these observations is demonstrated by previous works on plasma molecules showing prognostic associations with symptomatic hemorrhages in human CA, related to the same canonical pathways. Authors suggest careful tracking of the association of Btx and hemorrhage in CA patients.

Keywords: Botulinum toxin; Cavernous angioma; Cerebral cavernous malformation; Hemorrhage; MEKK; TLR.

Publication types

Case Reports
Review

MeSH terms

Adult
Botulinum Toxins, Type A / administration & dosage
Botulinum Toxins, Type A / adverse effects*
Brain Neoplasms / drug therapy*
Chronic Pain
Female
Hemangioma, Cavernous / drug therapy*
Humans
Intracranial Hemorrhages / etiology*
MAP Kinase Kinase Kinase 3 / metabolism
Magnetic Resonance Angiography
Migraine Disorders / etiology
Neurotoxins / administration & dosage
Neurotoxins / adverse effects*
Toll-Like Receptors / metabolism

Substances

Neurotoxins
Toll-Like Receptors
MAP Kinase Kinase Kinase 3
MAP3K3 protein, human
Botulinum Toxins, Type A
onabotulinum toxin A